A case of Adrenocoricotrophic hormone -independent bilateral adrenocortical macronodular hyperplasia concomitant with primary aldosteronism

Mao Tokumoto, Naoyoshi Onoda, Yukie Tauchi, Shinichiro Kashiwagi, Satoru Noda, Norikazu Toi, Masahumi Kurajoh, Masahiko Ohsawa, Yuto Yamazaki, Hironobu Sasano, Kosei Hirakawa, Masaichi Ohira

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2 Citations (Scopus)

Abstract

Background: Adrenocoricotrophic hormone (ACTH) - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) is a rare cause of Cushing's syndrome, and is characterized by bilateral adrenal hyperplasia. However, Primary aldosteronism (PA) is a relatively common adrenal disease. Case presentation: A 56-year-old man who has been treated hypertension and diabetes mellitus was detected low plasma potassium level with an elevated level of plasma aldosterone concentration and bilateral adrenal swelling. Endocrinological examinations showed autonomous secretion of cortisol and aldosterone, with suppression of plasma ACTH level and renin activity. A selective adrenal venous sampling demonstrated that left adrenal gland was responsible for aldosterone hypersecretion. He was diagnosed preclinical Cushing's syndrome due to ACTH - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) associated with aldosterone producing adenoma of the left adrenal gland. A laparoscopic left adrenalectomy was performed. Conclusion: The resected adrenal specimen histologically consisted with a diagnosis of AIMAH. Moreover, tiny cell clusters positive immunostaining for aldosterone synthase was revealed. This is a rare case of AIMAH accompanied by preclinical Cushing's syndrome and primary aldosteronism.

Original languageEnglish
Article number97
JournalBMC Surgery
Volume17
Issue number1
DOIs
Publication statusPublished - 2017 Sep 6

Keywords

  • ACTH - Independent bilateral adrenocortical macronodular hyperplasia
  • Laparoscopic unilateral adrenolectomy
  • Preclinical Cushing's syndrome
  • Primary aldosteronism

ASJC Scopus subject areas

  • Surgery

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    Tokumoto, M., Onoda, N., Tauchi, Y., Kashiwagi, S., Noda, S., Toi, N., Kurajoh, M., Ohsawa, M., Yamazaki, Y., Sasano, H., Hirakawa, K., & Ohira, M. (2017). A case of Adrenocoricotrophic hormone -independent bilateral adrenocortical macronodular hyperplasia concomitant with primary aldosteronism. BMC Surgery, 17(1), [97]. https://doi.org/10.1186/s12893-017-0293-z